Speech Outcome in Cleft Palate
Cleft lip and palate (CP±L) occurs in Sweden in about one child per 500 born. However, children with CLP are a heterogeneous group with several subgroups and differences between them. This applies to the type of cleft, size and cooccurrence of other malformations or disabilities.
Primary surgical repair of the cleft palate is primarily to provide the best conditions for speech development, occlusion and appearance. The treatment is multidisciplinary and also includes the speech and palatal function, the function of the ear and hearing, which are important contributing components for speech and language development, as well as dental development and growth in the face, which are important contributing components for appearance.
Early identification of children who are at risk of being delayed or deviating in speech development is necessary to be able to develop care programs and routines to prevent poor health in terms of speech disorders that make speech different and difficult to understand and that affect the ability for communicative activity and participation.
Speech deviations related to CP±LG
Today there is rather good knowledge of the occurrence of speech deviations related to cleft palate as well as about differences depending on the type of cleft. International studies show relatively consistent results, although these vary. In summary, up to 50 percent of the children treated for CP±L still seem to have speech and / or nasality problems in preschool or early school age, and some support that there is a higher incidence of deviations in speech and language abilities in the case of a major defect or in the presence of other malformations/syndromes.
Furthermore, that peers perceive deviations in speech which could have a negative impact on participation. Data on ear and hearing problems are still scarce, but provide support for the fact that otitis media with effusion (OME) with associated fluctuating mild hearing loss occurs in virtually all children with cleft palate. However, there is still no support for any surgical protocol to providing the best conditions for speech development. In addition, there is insufficient knowledge of people's own perception of their speech and the need for and effect of speech intervention.
Ongoing projects
The group investigates the effect of the method for surgery of the cleft palate in children born with cleft lip, jaw and / or cleft palate (CP±L) on how speech develops, how people perceive their own speech and the effect of speech therapy.
project, which started in 1997, consists of three concurrent randomized trials of primary surgery for infants born with complete unilateral cleft lip and palate (UCLP). The project was developed and executed by 10 North European cleft teams: Århus/Copenhagen (Denmark), Helsinki (Finland), Bergen/Oslo (Norway), Gothenburg/Linköping/ Stockholm (Sweden), and Manchester/Belfast (UK). One surgical protocol was defined to serve as a common method in each trial, against which a local protocol was compared. Recruitment of 448 infants took place over a 9-year period, with high subsequent retention of participants in each trial. The 5- and 10-year outcome has been published. Longitudinal analyses are ongoing as well as follow-up at young adulthood.
Trial registration number ISRCTN29932826.
PI Speech: Anette Lohmander 2001 – 2016, Christina Persson 2017 –
The primary objective of the -trial is to determine whether surgery for cleft palate performed at 6 or 12 months of age is most beneficial for speech outcomes. Approximately 550 children from five countries (all centres Denmark, Norway and Sweden, the Bauru center in Brazil and around 10 in the UK).
The primary outcome is perceived velopharyngeal function at 5 years of age. Secondary outcomes at 12 and 36 months of age will be reported as well.
Trial registration number NCT00993551.
PI Stockholm Center: Anette Lohmander.
Fluctuating hearing loss due to otitis media with effusion (OME) is very common in children born with cleft palate (CP±L). In the TUTH project, early development of speech and hearing is studied in a controlled group study of children with CP±L and children with OME, respectively.
Subprojects include hearing, perception and production of consonant sounds, consonant inventory and proficiency, velopharyngeal function, intelligibility, vocabulary and language comprehension up to 3 years of age.
PI: Anette Lohmander
Postdoc-project: Ã…sa Okhiria
In Sweden, children born with cleft palate are operated on at 6 different university hospitals. There are mainly two methods of cleft palate surgery that are used; closure in one session at 9-12 months of age or closure in two sessions where the soft palate is closed at around 6 months and the cleft in the hard palate at around 2 years of age (earlier in preschool or early school age). The evidence regarding the speech result after various surgical methods has been insufficient both in terms of a specific age and longitudinally. The purpose of a series of projects is partly to contribute generally to the basis for choosing a surgical treatment method by evaluating how speech develops in children born with LKG cleft, and partly to specifically investigate the impact of speech abnormalities on the environment's perception of the individual's speech and communication. Three project parts have been completed:
- Speech outcome in children born with cleft lip and palate: aspects of function and environment (doctoral student Jill Nyberg, 2016, PhD).
- Orofacial function, articulation proficiency and intelligibility in 5-year old children born with cleft lip and palate (doctoral student Ann Malmenholt, 2020: PhD)
- Speech outcome and self-reported communicative ability in young adults born with unilateral cleft lip and palate: comparing long-term results after two different surgical methods for palatal repair (doctoral student Petra Peterson, 2022; PhD)
The speech of a group of children born with UCLP and treated with a two-stage palate repair are examined longitudinally from 1-7 years and is compared with the results in a previous group treated with one-stage palate repair. Age-relevant speech variables are assessed perceptually, and the results are compared between the groups. Both studies include measures of intelligibility, that is, how well the environment perceives / understands the speech, and the individuals' own perception of their speech.
Doctoral student: Emilie Hagberg
Speech disorders in children born with cleft palate are treated with speech therapy, but treatment studies of speech disorders related to cleft palate are still few and with great methodological variation. The project includes a systematic review and meta-analysis.
Furthermore, two treatment studies where both are controlled case series (Controlled single subject series design) in collaboration with speech and language pathologists in the Stockholm Region. In the second, distance treatment is planned.
Postdoctoral project: Anders Sand
Doctoral student project: Emilie Hagberg
These and the other projects are described on the respective researcher's profile page.
Collaborators
Anette Lohmander
Project leaderEmilie Hagberg
̽»¨¾«Ñ¡, Karolinska UniversitetssjukhusetPetra Peterson
Karolinska UniversitetssjukhusetAnn Malmenholt
Karolinska Universitetssjukhuset, ̽»¨¾«Ñ¡Jill Nyberg
MentorÃ…sa Okhiria
Affiliated to ResearchChristina Persson
Göteborgs universitetKristina Klintö
Malmö Universitetssjukhus/Lunds universitetDissertations
Petra Peterson, PhD, KI, 2022: .
Birgitta Tengroth, Med Lic, KI, 2020: .
Ann Malmenholt, PhD, KI, 2020: .
Jill Nyberg, PhD, KI, 2016: .
Kristina Klintö, PhD, KI, 2014: .
Selected publications
Scandcleft Project
Persson C, Davies J, Havstam C, Søgaard H, Bowden M, Boers M, Nielsen JB, Alaluusua S, Lundeborg Hammarström I, Emborg BK, Sand A, Lohmander A. Speech outcome at 5 and 10 years in unilateral cleft lip and palate: influence of speech therapy and secondary velopharyngeal surgery. Cleft Palate-Craniofac J. Accepted Dec 20, 2023.
Willadsen E, Jørgensen L, Alalusuua S, Pedersen NH, Nielsen J, Hölttä E……Lohmander A, Persson C. Int J Lang Comm Dis. 2023;58:892-909.
Persson C, Pedersen NH, Hayden C, Bowden M, Aukner R, Vindenes H, Ã…byholm F, Whitby D, Willadsen E, Lohmander A. . Cleft Palate-Craniofac J. 2020;57:352-363.
Hammarström IL, Nyberg J, Alaluusua S, Rautio J, Neovius E, Berggren A, Willadsen E, Persson C, Lohmander A. . Cleft Palate-Craniofac J. 2020;57:458-469.
Willadsen, E., Lohmander, A., Persson, C., Boers, M., Kisling-Møller, M., Havstam, C., . . . Andersen, M. (2019). . Cleft Palate Craniofac J, 2019;56:1276-1286.
Lohmander A, Persson C, Willadsen E, Lundeborg I, Alaluusua S, Aukner R, et.al. . J Plast Surg Hand Surgery, 2017;51:26-36.
Willadsen E, Lohmander A, Persson C, Lundeborg I, Alaluusua S, Aukner R, et.al. . J Plast Surg Hand Surgery, 2017;51:37-50.
Lohmander A, Hagberg E, Persson C. Willadsen E, LundeborgI, Davies J, Havstam C, Boers M, Kisling-Møller M, Alaluusua S, AuknerR, Pedersen NH, Tuurunen L, NybergJ. . Clinical Linguistics & Phonetics, 2017;31:589-597.
Billaud Feragen K, Semb G, Heliövaara A, Lohmander A, Johannessen EC, Boysen BM, et al. . J Plast Surg Hand Surg, 2017;51:80-86.
Semb G, Enemark H, Friede H, Paulin G, Lilja J, Rautio J, Andersen M, Åbyholm F, Lohmander A, ….Worthington W. S. J Plast Surg Hand Surg, 2017;51:1-12.
Lohmander A, Willadsen E, Bowden M, Henningsson G, Persson C, Hutters B. . Cleft Palate-Craniofac J. 2009; 46:347-362.
TOPS-project
Gamble C, Persson C, Willadsen E, ………..Lohmander A, ………..Nyberg J, ……….Westberg LR, ……..Munro K, Walsh T, Shaw W. . New England Journal of Medicine, 2023;389(9):795-807.
Shaw W, Semb G, Lohmander A, Persson C, Willadsen E, Clayton-Smith J, Trindade IK, Munro KJ, Gamble C, Harman NH, Conroy EJ, Weichart D, Williamson P. T. British Medical Journal Open, 2019;9:e029780.
Speech outcome and communicative ability in individuals born with CP±L - comparing results after different methods for palatal repair in Sweden
Peterson P, Nyberg J, Persson C, Mark H, Lohmander A. . Cleft Palate-Craniofac J. 2022;59(6):751–764.&²Ô²ú²õ±è;
Klintö K, Brunnegård K, Havstam C, Appelqvist M, Hagberg E, Taleman A-S, Lohmander A. . J Plast Surg Hand Surgery, 2019;53:309-315.
Malmenholt, A, McAllister A, Lohmander A. . Cleft Palate-Craniofacial J. 2019;56:321-330.
Nyberg J, Neovius E, Lohmander A. . J Plastic Surgery Hand Surgery, 2018;52:20-29.
Nyberg J, Peterson P, Lohmander A. . Int J Pediatric Otorhinolaryngology, 2014;78:1662-1670.
Lohmander A, Friede H, Lilja J. . Cleft Palate-Craniofac J, 2012;49:657-671.
Lohmander A, Persson C. . Cleft Palate-Craniofacial J, 2008;45:32-41.
Lohmander A, Friede H, Elander A, Persson C, Lilja J. . Scand J Plast Reconstr Surg Hand Surgery, 2006;46:267-274.
Early Development of Speech and Hearing – TUTH (and related)
Lohmander A, Raud Westberg L, Olsson S, Tengroth B, Flynn T. . Cleft Palate-Craniofac J. 2021;58:894-905.
Tengroth B, Lohmander A, Hederstierna C. . Cleft Palate Craniofac J. 2020;57:616-623.
Flynn T, Lohmander A. . Otology & Neurology, 2014;35:989-996.
Flynn T, Möller C, Jönsson R, Lohmander A. . Int J of Pediatric Otorhinolaryngology, 2009;73:1441-1446.
Effect of speech therapy of LKG-related speech disorders
Hagberg E, Andersson Stockhaus U, Fagius T, Hartstein M, Raud Westberg L, Nyberg J, Sand A, Lohmander A: Speech therapy in children with cleft palate: speech outcome from multiple single subject casesIJSLP Under Review 2024.
Sand A, Hagberg E, Lohmander A. . J Speech Lang Hear Res. 2022;65:555-573.
Financing
Forte
Grants from the Swedish government under the ALF agreement
NIH, National Institute for Dental and Craniofacial research (NIH-NIDCR)
Stiftelsen Frimurare Barnhuset i Stockholm
Aina Börjesons Research Foundation
Queen Silvia's Jubilee Fund